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489 Abstract: We describe a rare case of large congenital lipoma in the upper lip of a six-month-old infant. Surgical excision of the tumor was successful, with satisfactory esthetic and functional results. (J Oral Sci 51, 489-491, 2009) Keywords: mouth neoplasms; adipose tissue; lipoma; congenital. Introduction Lipoma is the most common benign mesenchymal tumor and can develop in any site where adipose tissue is present. It mainly occurs in subcutaneous tissue, but can also present in deeper regions. Peak occurrence is mainly in the fifth or sixth decades of life, and the tumor is uncommon in childhood (1). Although 15 to 20% of these tumors occur in the head and neck region, only 1 to 4% affect the oral cavity, representing 0.1 to 5% of all benign tumors of the mouth (2). This report describes a rare case of large congenital lipoma in the upper lip of a six-month-old infant. Case Report A six-month-old girl was referred to the Department of Oral and Maxillofacial Surgery. Her mother had noticed increased swelling of the child’s upper lip. A mass of soft tissue had been observed in the upper lip at birth, but no treatment was performed at that stage. The mass appeared to be gradually enlarging. The mother also reported difficulties in breastfeeding the child, as the tumor rendered lip seal to the breast impossible. Maxillofacial examination revealed an increased volume of the upper lip; the mass had a soft consistency, well-defined edges, no pulsation, and was symptomatic upon palpation (Fig. 1A). The lesion had a sessile base and the overlying mucosa appeared normal. Due to the large dimensions of the tumor, the child exhibited lip incompetence, with dryness of the oral mucosa and a mouth-breathing pattern. No radiographic abnormalities were observed. The proposed treatment was excision biopsy under general anesthesia. Aspiration was first performed, which was negative (Fig. 1B). A surface incision was made in the labial mucosa, followed by submucosal dissection. This clearly showed a yellowish mass, which could be well delimited and excised after exposure (Fig. 1C and 2A). The excised tumor (Fig. 2B) was sent for histopathological analysis, the result of which was lipoma (Fig. 2C). The child has been followed up for two years, exhibiting satisfactory esthetics and function, with no signs of recurrence (Fig. 2D). Journal of Oral Science, Vol. 51, No. 3, 489-491, 2009 Correspondence to Dr. Hécio Henrique Araújo Morais, Faculdade de Odontologia de Pernambuco – FOP/UPE, Av. General Newton Cavalcanti, 1650, Tabatinga, 54753-220, Camaragibe – Pernambuco, Brazil Tel: +55-81-88868677 Fax: +55-81-34582867 E-mail: [email protected] Congenital lipoma of the lip: a case report Hécio H. A. Morais 1,2) , André Vajgel 3) , Nelson S. Rocha 1) , Ricardo W. F. Carvalho 4) , Antonio F. Caubi 4) and Ricardo J. H. Vasconcellos 4) 1) Doctorate Program in Oral and Maxillofacial Surgery, Department of Oral and Maxillofacial Surgery, Pernambuco School of Dentistry, University of Pernambuco, Recife, Brazil 2) Department of Oral and Maxillofacial Surgery, School of Dentistry, Rio Grande do Norte State University, Caicó, Brazil 3) Program of MsC in Oral and Maxillofacial Surgery, Department of Oral and Maxillofacial Surgery, Pernambuco School of Dentistry, University of Pernambuco, Recife, Brazil 4) Department of Oral and Maxillofacial Surgery, Oswaldo Cruz Hospital and Pernambuco School of Dentistry, University of Pernambuco, Recife, Brazil (Received 8 January and accepted 6 June 2009) Case Report

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489

Abstract: We describe a rare case of large congenitallipoma in the upper lip of a six-month-old infant.Surgical excision of the tumor was successful, withsatisfactory esthetic and functional results. (J Oral Sci51, 489-491, 2009)

Keywords: mouth neoplasms; adipose tissue; lipoma;congenital.

IntroductionLipoma is the most common benign mesenchymal tumor

and can develop in any site where adipose tissue is present.It mainly occurs in subcutaneous tissue, but can alsopresent in deeper regions. Peak occurrence is mainly inthe fifth or sixth decades of life, and the tumor is uncommonin childhood (1). Although 15 to 20% of these tumors occurin the head and neck region, only 1 to 4% affect the oralcavity, representing 0.1 to 5% of all benign tumors of themouth (2).

This report describes a rare case of large congenitallipoma in the upper lip of a six-month-old infant.

Case ReportA six-month-old girl was referred to the Department of

Oral and Maxillofacial Surgery. Her mother had noticedincreased swelling of the child’s upper lip. A mass of softtissue had been observed in the upper lip at birth, but notreatment was performed at that stage. The mass appearedto be gradually enlarging. The mother also reporteddifficulties in breastfeeding the child, as the tumor renderedlip seal to the breast impossible. Maxillofacial examinationrevealed an increased volume of the upper lip; the masshad a soft consistency, well-defined edges, no pulsation,and was symptomatic upon palpation (Fig. 1A). The lesionhad a sessile base and the overlying mucosa appearednormal. Due to the large dimensions of the tumor, thechild exhibited lip incompetence, with dryness of the oralmucosa and a mouth-breathing pattern. No radiographicabnormalities were observed. The proposed treatment wasexcision biopsy under general anesthesia. Aspiration wasfirst performed, which was negative (Fig. 1B). A surfaceincision was made in the labial mucosa, followed bysubmucosal dissection. This clearly showed a yellowishmass, which could be well delimited and excised afterexposure (Fig. 1C and 2A). The excised tumor (Fig. 2B)was sent for histopathological analysis, the result of whichwas lipoma (Fig. 2C). The child has been followed up fortwo years, exhibiting satisfactory esthetics and function,with no signs of recurrence (Fig. 2D).

Journal of Oral Science, Vol. 51, No. 3, 489-491, 2009

Correspondence to Dr. Hécio Henrique Araújo Morais, Faculdadede Odontologia de Pernambuco – FOP/UPE, Av. General NewtonCavalcanti, 1650, Tabatinga, 54753-220, Camaragibe –Pernambuco, BrazilTel: +55-81-88868677Fax: +55-81-34582867E-mail: [email protected]

Congenital lipoma of the lip: a case report

Hécio H. A. Morais1,2), André Vajgel3), Nelson S. Rocha1), Ricardo W. F. Carvalho4),Antonio F. Caubi4) and Ricardo J. H. Vasconcellos4)

1)Doctorate Program in Oral and Maxillofacial Surgery, Department of Oral and Maxillofacial Surgery,Pernambuco School of Dentistry, University of Pernambuco, Recife, Brazil

2)Department of Oral and Maxillofacial Surgery, School of Dentistry, Rio Grande do Norte State University,Caicó, Brazil

3)Program of MsC in Oral and Maxillofacial Surgery, Department of Oral and Maxillofacial Surgery,Pernambuco School of Dentistry, University of Pernambuco, Recife, Brazil

4)Department of Oral and Maxillofacial Surgery, Oswaldo Cruz Hospital and Pernambuco School of Dentistry,University of Pernambuco, Recife, Brazil

(Received 8 January and accepted 6 June 2009)

Case Report

490

DiscussionCongenital lipoma in the oral cavity is a rare entity and

its appearance in the lip is even rarer. Few cases of con-genital lipoma are reported in the literature and none werefound regarding lipoma affecting the lip. De Carvalho etal. (3), Dimitrakopoulos et al. (4), and Mahabir et al. (5)reported congenital lipoma in the vestibular fornix of aseven-month-old boy, the tongue of a 20-day-old girl andthe soft palate of an eight-month-old boy, respectively.

The histogenesis of this lesion remains unclear. A studyof the embryogenesis of fat tissue reveals that it appearsin the embryo, and the formation of new lobules ceasesin late fetal life or the early postnatal period (6). Lipomais thought to result from a continuation of the proliferationof these fat tissue lobules.

The distinction between benign neoplasm, malformation,and hyperplasia may not be clinically clear. Lipoma inadults is commonly considered a neoplasm, whereas inchildren it is classified as either a neoplasm or malformation(7). Solitary lipomas, such as those found in the presentcase, are considered true neoplasms rather than develo-pmental malformations.

Clinically, lipomas are generally mobile, painless,submucosal nodules with a yellowish color; thesecharacteristics were all noted in the congenital variantdescribed here. Due to these clinical characteristics, otherlesions should be considered in the differential diagnosisof oral lipoma, such as dermoid and epidermoid cysts andcongenital lip entities, including common vascular lesions(hemangioma and lymphangioma), benign mesen-chymoma, and mucous cysts (8). However, these lesionsmay occur at other sites of the oral mucosa. Mesenchymaltumors should also be included in the differential diagnosis(9).

It is unusual for children to have classic lipomas;lipoblastoma and lipoblastomatosis are more oftendiagnosed in pediatric patients (1). Given its congenitalnature, lipoblastoma, although rare, should also be includedin the histological differential diagnosis. Thus, a discerningclinical diagnosis and histological analysis is important fordiagnostic confirmation.

Congenital lipomas have been reported but are rare(7,9), and in some, familial predisposition (7) has beensuggested; however this was not indicated in the presentcase. Congenital lipoma is commonly described inassociation with craniofacial anomalies. For example,congenital lipoma was described in an uncommon case oforal-facial-digital syndrome, differing from the standardpattern by exhibiting congenital lipoma rather than thehamartoma of the tongue normally described. The authorsdescribed this as a variant of type II oral-facial-digitalsyndrome (7).

Congenital lipoma is an extremely rare benign lipomatoustumor. The case described here exhibited the classicfeatures of this condition. Surgical excision of the tumorwas successful, with the child exhibiting satisfactoryesthetics and function, with no signs of recurrence.

References1. Furlong MA, Fanburg-Smith JC, Childers EL (2004)

Fig. 2 (A) Surgical site; (B) Excised tumor; (C) Histologicalappearance; (D) 2 years after operation.

Fig. 1 (A) Frontal view of the lesion; (B) Negative aspiration;(C) Exteriorization of the tumor.

491

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2. Fregnani ER, Pires FR, Falzoni R, Lopes MA,Vargas PA (2003) Lipomas of the oral cavity: clinicalfindings, histological classification and proliferativeactivity of 46 cases. Int J Oral Maxillofac Surg 32,49-53.

3. Dimitrakopoulos I, Zouloumis L, Trigonidis G(1990) Congenital lipoma of the tongue. Report ofa case. Int J Oral Maxillofac Surg 19, 208.

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5. Mahabir RC, Mohammad JA, Courtemanche DJ(2000) Lipoma of the cleft soft palate: a case reportof a rare congenital anomaly. Cleft Palate Craniofac

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7. Ghossaini SN, Hadi U, Tawil A (2002) Oral-facial-digital syndrome type II variant associated withcongenital tongue lipoma. Oral Surg Oral Med OralPathol Oral Radiol Endod 94, 324-327.

8. Bandéca MC, de Pádua JM, Nadalin MR, OzórioJEV, Silva-Sousa YTC, da Cruz Perez DE (2007)Oral soft tissue lipomas: a case series. J Can DentAssoc 73, 431-434.

9. Yonezawa H, Harada K, Enomoto S (2000)Congenital lipomatoid mass of the tongue. Int JOral Maxillofac Surg 29, 138-139.